TY - JOUR
T1 - Isavuconazole in a Successful Combination Treatment of Disseminated Mucormycosis in a Child with Acute Lymphoblastic Leukaemia and Generalized Haemochromatosis
T2 - A Case Report and Review of the Literature
AU - Pomorska, Anna
AU - Malecka, Anna
AU - Jaworski, Radoslaw
AU - Radon-Proskura, Julia
AU - Hare, Rasmus Krøger
AU - Nielsen, Henrik Vedel
AU - Andersen, Lee O'Brian
AU - Jensen, Henrik Elvang
AU - Arendrup, Maiken Cavling
AU - Irga-Jaworska, Ninela
PY - 2019/2
Y1 - 2019/2
N2 - Invasive mucormycosis in immunocompromised children is a life-threatening fungal infection. We report a case of a 7-year-old girl treated for acute lymphoblastic leukaemia complicated by disseminated mucormycosis during induction therapy. Microscopic examination of surgically removed lung tissue revealed wide, pauci-septate hyphae suggesting a Mucorales infection. This diagnosis was confirmed immunohistochemically and by PCR analysis followed by a final identification of Cunninghamella sp. The patient was treated successfully with surgical debridement and antifungal combination therapy with amphotericin B, caspofungin and isavuconazole. The use of isavuconazole in a child was not previously reported. Additionally, case reports concerning pulmonary mucormycoses in paediatric population published after 2010 were reviewed. Nineteen out of 26 identified patients suffered from haematological diseases. Reported mortality reached 38.5%. By the fact of rising morbidity, unsatisfactory results of treatment and remaining high mortality of mucormycoses in immunocompromised patients, new therapeutic options are warrant. Isavuconazole, with its broad-spectrum activity, good safety profile and favourable pharmacokinetics, is a promising drug. However, further studies are necessary to confirm positive impact of isavuconazole on mucormycosis treatment in children.
AB - Invasive mucormycosis in immunocompromised children is a life-threatening fungal infection. We report a case of a 7-year-old girl treated for acute lymphoblastic leukaemia complicated by disseminated mucormycosis during induction therapy. Microscopic examination of surgically removed lung tissue revealed wide, pauci-septate hyphae suggesting a Mucorales infection. This diagnosis was confirmed immunohistochemically and by PCR analysis followed by a final identification of Cunninghamella sp. The patient was treated successfully with surgical debridement and antifungal combination therapy with amphotericin B, caspofungin and isavuconazole. The use of isavuconazole in a child was not previously reported. Additionally, case reports concerning pulmonary mucormycoses in paediatric population published after 2010 were reviewed. Nineteen out of 26 identified patients suffered from haematological diseases. Reported mortality reached 38.5%. By the fact of rising morbidity, unsatisfactory results of treatment and remaining high mortality of mucormycoses in immunocompromised patients, new therapeutic options are warrant. Isavuconazole, with its broad-spectrum activity, good safety profile and favourable pharmacokinetics, is a promising drug. However, further studies are necessary to confirm positive impact of isavuconazole on mucormycosis treatment in children.
KW - Amphotericin B/administration & dosage
KW - Antifungal Agents/administration & dosage
KW - Caspofungin/administration & dosage
KW - Child
KW - Cunninghamella/isolation & purification
KW - Debridement
KW - Drug Therapy, Combination/methods
KW - Drug-Related Side Effects and Adverse Reactions/epidemiology
KW - Female
KW - Hemochromatosis/complications
KW - Humans
KW - Invasive Fungal Infections/diagnosis
KW - Mucormycosis/diagnosis
KW - Nitriles/administration & dosage
KW - Precursor Cell Lymphoblastic Leukemia-Lymphoma/complications
KW - Pyridines/administration & dosage
KW - Treatment Outcome
KW - Triazoles/administration & dosage
U2 - 10.1007/s11046-018-0287-0
DO - 10.1007/s11046-018-0287-0
M3 - Review
C2 - 30039238
SN - 0301-486X
VL - 184
SP - 81
EP - 88
JO - Mycopathologia
JF - Mycopathologia
IS - 1
ER -